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Dr Dennis Nyuk Fung Lim<sup>1</sup>, Dr Andrew Steel<sup>2</sup> Adenocarcinoma in EIPD
Dr Dennis Nyuk Fung Lim1, Dr Andrew Steel2, 17 November 2014
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1 Specialty Training Registrar ST5
Digestive Disease Centre
University Hospital of Leicester NHS Trust
Leicester Royal Infirmary
Infirmary Square
Leicester
LE1 5WW
United Kingdom

2 Consultant Gastroenterologist
Department of Gastroenterology and Hepatology
Kettering General Hospital NHS Trust
Rothwell Road
Kettering
NN16 8UZ
United Kingdom

Summary:

Oesophageal intramural pseudodiverticulosis (EIPD) is a rare condition of unknown aetiology characterised by multiple, flask-shaped out-pouching with segmental or diffuse involvement of the oesophagus.  EIPD has been associated with oesophageal stricture, candida oesophagitis and oesophageal dysmotility.  Our knowledge of long term outcome of this condition is limited.  We report a case of adenocarcinoma in a patient with EIPD that had multiple oesophageal dilatations

 

Case report:

A 63 year old man with Type 2 diabetes was diagnosed of oesophageal intramural pseudo-diverticulosis EIPD with recurrent high oesophageal stricture since 2007.  He was treated successfully with wire-guided through the scope balloon dilatations (inflated up to 10mm).  Multiple oesophageal biopsies showed evidence of active chronic inflammatory changes with fungal hyphae on Periodic acid-Schiff stain.

See Figs 1, 2 and 3.

Fig 1: Barium swallow showed multiple “flash-shaped” outpouchings involving the whole of the oesopghagus typical of EIPD

Fig 2: Upper gastro-intestinal endoscopy showed oesophageal intramural pseudo-diverticulosis with stricture requiring wire-guided through the scope balloon dilatation

Fig 3: Oesophageal biopsy showed active chronic inflammatory changes with fungal hyphae on Periodic acid-Schiff stain. No evidence of dysplasia or malignancy identified

He received oral fluconazole for the treatment of oesophageal candidiasis as well as long term full dose oral proton pump inhibitor.  He remained asymptomatic for several years.  Patient re-presented with dysphagia in 2013.  A diagnostic upper GI endoscopy showed stricture at the gastro-oesophageal junction (GOJ) 40cm from incisor.  The stricture was unsuccessfully treated with wire-guided through the scope balloon dilatations (inflated up to 12mm).  Multiple biopsies confirmed adenocarcinoma of tubular intestinal type.  Positron emission tomography (PET), Computerised tomography (CT) and Endoscopic Ultrasound scan(EUS) staging showed T3,N0,M0.  The patient received pre-operative adjuvant chemotherapy followed by radical total oesophagectomy.

See Figs 4 and 5.

Fig 4: UGI endoscopy showed stricture at gastro-oesophageal junction (GOJ) unsuccessfully treated with balloon dilatation

Fig 5: Intense uptake in the distal oesophageal lesion which extended from 40-45cm

 

Discussion:

Since first reported EIPD in 1960, there were around 200 cases published world-wide.1,2 Up to 90% of patients with EIPD have associated oesophageal stenosis of various levels most commonly in the upper oesophagus secondary to chronic stasis oesophagitis.3,4  EIPD is often regarded as benign oesophageal disease but there are several reports where EIPD was found in patients with oesophageal carcinoma. 5   The aetiology behind EIPD developing oesophageal cancer could be due to metaplastic changes of the squamous epithelium within the excretory ducts of oesophageal submucosal glands in EIPD. 6 The pre-malignant nature of metaplastic epithelium is widely recognised in the upper gastro-intestinal tract.  The increased prevalence of EIPD in patients with oesophageal carcinoma may warrant periodic surveillance in this small population of patients.

 

References:

1.      Mendl K, Tanner CH. Intramural diverticulosis of the oesophagus and Rokitansky-Aschoff sinuses in the gall-bladder. Br J Radiol. 1960;33:496-501.

2.      Levine MS, Moolten DN, Herlinger H, Laufer I. Esophageal intramural pseudodiverticulosis: a reevaluation.  Am J Roentgenol. 1986;147:1165-1170.

3.      Sabanathan S, Salama FD, Morgan WE. Oesophageal intramural pseudo-diverticulosis. Thorax 1985;40:849-857.

4.      Cho SR, Sanders MM, Turner MA, Liu CI, Kipreos BE. Esophageal intramural pseudodiverticulosis. Gastrointest Radiol 1981;6:9-16.

5.       Plavsic BM, Chen MY, Gelfand DW, Drnovsek VH, Williams JP 3rd, Kogutt MS, Terry JA, Plenkovich D. Intramural pseudodiverticulosis of the esophagus detected on barium esophagograms: increased prevalence in patients with esophageal carcinoma. Am J Roentgenol 1995;165:1381-1385.

6.      Kataoka H, Higa T, Koono M. An autopsy case report of diffuse esoophageal intramural pseudodiverticulosis. Acta Pathol Jpn 1992;42:837-840.

 
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